Interventions to build resilience in family carers of people living with dementia: a systematic review protocol Andrea Petriwskyj 1 Deborah Parker 1 Siobhan O Dwyer 2 Wendy Moyle 2 Nikki Nucifora 1 1. The University of Queensland/Blue Care Research and Practice Development Centre and the Australian Centre for Evidence Based Community Care: a Collaborating Center of the Joanna Briggs Institute, The University of Queensland, Australia 2. Centre for Health Practice Innovation, Griffith University, Queensland, Australia Corresponding author Andrea Petriwskyj a.petriwskyj@uq.edu.au Review question/objective The objective of this review is to examine the existing evidence and knowledge regarding interventions for building resilience in family carers of people living with dementia. More specifically, the review questions to be addressed by quantitative studies are: 1. How effective are the existing interventions? 2. What factors moderate or influence the effectiveness of the existing interventions? Review questions to be addressed by qualitative studies are: 3. What are family members experiences or perceptions of the existing interventions? 4. What factors influence the implementation of the existing interventions? Background In 2010, 36 million people worldwide were estimated to have dementia and that number is predicted to double every 20 years, to more than 115 million in 2050. 1 While family carers currently provide the cornerstone of care for people with dementia, the demand for family carers is expected to exceed supply by 2029. 2 Caring for a person with dementia is considered to be particularly demanding due to the combination of physical, cognitive and behavioral symptoms the person with dementia may exhibit. Additionally, family carers are required to complete a variety of daily tasks that are often physically, emotionally, socially and financially demanding. 3 The negative health consequences for family caregivers of people with dementia, particularly with regard to their mental health, have been well doi: 10.11124/jbisrir-2015-1994 Page 44
documented in recent years. 4,5 Recent studies have indicated that family carers of people with dementia have higher rates of depression, anxiety and hopelessness than their non-caring counterparts, as well as lower subjective wellbeing. 6-8 As a group they report experiencing high levels of burden, stress and distress, 9-12 contemplate suicide at more than eight times the rate of the general population, 6,13 and are generally considered to have an increased risk of mortality. 14 Not all carers, however, succumb to the negative effects of caring. Carers who are able to recover from, resist, or adapt to the physical and psychological demands of caring can be considered resilient. 15-17 Resilience is broadly defined as the process of surviving adversity, returning to previous levels of functioning (recovering) and achieving positive outcomes as a result of the experience (thriving). 16-19 Resilience has been found to be inversely related to depression, anxiety, psycho-active drug use and perceived burden, and to be positively related to health and wellbeing. 9,20-22 It is also considered an important factor in suicide prevention. 23,24 In addition to the reduction in the negative effects of dementia caregiving, resilience has also been found to be positively associated with a number of factors that promote positive outcomes such as self-efficacy, self-esteem, problem focused coping, mastery, flexibility and adaptation. 9,21,22 Resilience in carers has also been found to predict the longevity of at-home care and is considered a protective factor against transition into institutionalized care. 21 In developed nations like Australia, the social and political agenda is clearly focused on keeping people with dementia in the community 25,26 and interventions that enhance resilience in family carers may go some way toward realizing this goal. Thus, resilience is considered to be an important positive psychological resource that can be strengthened to promote positive outcomes in dementia family caregiving. 27 Although some authors argue that resilience is a personality trait, there is growing support for the view that this is a psychological process that is amenable to change. 27,28 The factors that promote it are considered to span biological, personal, social, cultural and environmental domains. 16-19 Biological factors include brain size, reuptake of neurotransmitters, and stress hormones such as cortisol and dehydroepiandrosterone. 16 Personal, social and cultural factors include personality traits, coping styles, social support, positive interpersonal relationships, and access to education and employment. 16,18 Environmental factors include stability, quality neighborhoods, and the absence of violence, trauma and mistreatment. 16,18 There are therefore a wide range of factors that may be the target of interventions to support or enhance resilience in family carers. There is limited research focused specifically and exclusively on interventions which treat resilience as a unique construct. Indeed, an initial search of literature limited to the use of this term in the title and abstract identified only two interventions; these examined the potential of an antidepressant drug, escitalopram, 3 and a poetry writing intervention 27 in enhancing resilience in these carers. Furthermore, given its relative infancy as a topic in caregiving research, there is a lack of consistency in how the concept of resilience has been defined and used in the literature. Some studies, for example, have referred to resilience in relation to positive factors such as confidence in caregiving, problem-solving skills, a strong sense of religion or spirituality and social support. 29 Other studies, on the other hand, have related resilience to a number of personality traits, such as flexibility, positivity, adaptability and self-sufficiency. 30 Similarly, whilst some advocate specific measures of resilience, others consider constructs with conceptual overlap such as self-efficacy, locus of control, perceived burden, psychological wellbeing, strength, coping, positive adjustment and resourcefulness. 11,21,31,32 Given that higher levels of resilience have been found to be strongly associated with a number of psychological constructs it is not unusual for them to be included as proxy measures for resilience within the literature. doi: 10.11124/jbisrir-2015-1994 Page 45
While little intervention research has targeted building resilience directly, there are a number of studies that have focused on such proxy measures. For example, several studies have focused on increasing coping, mastery, self-efficacy and competence whilst others have been aimed at reducing stress, perceived burden and depression, and anxiety symptomatology. The majority of interventions relating to these proxy measures broadly fall into eight categories: psychotherapy, psychoeducational, supportive groups, training, pharmacotherapy, multicomponent interventions and other forms (i.e. meditation, telephone groups, etc.). 33,34 A meta-analysis conducted by Pinquart and Sorensen 33 analyzing 127 interventions with dementia family caregivers found significant effects on subjective burden, depression symptoms, subjective wellbeing (SWB), self-efficacy/knowledge and symptoms of the care recipient. Furthermore, intervention effects were found to be ongoing for burden, depressive symptoms and self-efficacy/knowledge after an average time lapse of 11 months. 33 Overall, the metaanalysis found psychoeducation interventions that provide active participation of caregivers (i.e. role playing and homework) to have the broadest effects across all outcomes. On the other hand, psychoeducational interventions that provide information only were found to have minimal impact across all variables except on caregiver knowledge. Cognitive behavioral therapy (CBT) interventions were found to dramatically reduce depression symptoms as well as caregiver burden. No significant effects were found for SWB or self-efficacy/knowledge; however the authors noted that these results should be interpreted with caution due to only one and three studies, respectively, examining these variables. Counselling interventions were found to significantly reduce burden and SWB, whilst supportive interventions were found to improve SWB. Structured multicomponent interventions were found to significantly reduce the level of institutionalization. An earlier meta-analysis involving the same authors yielded similar results as well as identifying a number of additional moderating factors. 34 The intervention setting (i.e. group versus individual) and duration (short versus long), as well as the caregiver age, gender, initial burden and type of caregiver-care receiver relationship (spouse versus child), were all found to moderate the effectiveness of interventions. Thus, a broader focus on proxy and related measures to resilience suggests that a number of interventions may have positive effects for carers. Whilst some studies relate the impact of their interventions on resilience specifically, not all of them provide this clarification, making it challenging to identify the extent to which such interventions enhance resilience. Literature addressing resilience is therefore somewhat varied in its focus and scope, and in its measurement of key constructs, making it challenging to identify effective interventions and potential methods to support or enhance resilience in this population. While much research has focused on the internal resources, characteristics and factors that facilitate resilience in carers, 15 and although reviews addressing broader constructs relating to resilience and wellbeing have been conducted previously, 33,34 to the knowledge of the authors a systematic review of interventions designed to build resilience among family carers of people with dementia has not been conducted. The use of proxy constructs has provided a broader body of evidence that suggests that there is the potential for a range of interventions to have a positive effect for family carers; however, the evidence specifically relating to resilience in family carers can be challenging to separate from this broader literature. This review therefore aims to identify and synthesize evidence regarding interventions for family carers of people with dementia that have enhancement of resilience as an aim. Keywords Dementia; family carers, resilience, interventions doi: 10.11124/jbisrir-2015-1994 Page 46
Inclusion criteria Participants This review will consider studies that include family carers of people with dementia. For the purpose of this review, the definition of dementia is not restricted to any specific type, stage or degree of severity and will be based on reporting in the original paper, regardless of whether a doctor s diagnosis is reported; however, papers that specify a focus only on mild cognitive impairment without specifying a substantive focus on dementia will be excluded. There is no age limit on participants, and family carers may include any and all family members involved in providing care to people with dementia, including but not limited to spouses and children. As the definition of family carers used in dementia caregiving literature is broad, this review will similarly adopt a definition, used by Palliative Care Australia and adopted from the Canadian Palliative Care Association that includes those who are closest to the patient in knowledge, care and affection. The family may include the biological family, the family of acquisition (related by marriage/contract), and the family of choice and friends (including pets). 35(p10) Thus, family carers may not necessarily be biologically related to the person with dementia. The review will exclude studies focusing primarily or substantially on paid carers. Only papers for which family carer data can be extracted separately will be included. The review will focus broadly on family carers, and will not require that the family carer being referred to is the primary carer of the person with dementia. Studies conducted in all settings will be included. Intervention This review will consider studies that investigate interventions to build resilience in family carers. For the purpose of this review an intervention refers to a method of interfering with the outcome of a condition or process and may include pharmacological, social, psychological, educational, carerelated and physical programs or treatments. Interventions will cover those provided by professionals as well as self-help strategies. Intervention comparators may include, but not be limited to, no treatment control, treatment as usual, waitlist control, comparison with alternative intervention/s, comparison with different medium of delivery (e.g. face-to-face versus via telephone) and comparisons of different frequencies and durations of intervention. Interventions directed towards the person with dementia themselves, such as training or behavior management interventions, will be excluded. Resilience will be defined as a psychological process enabling carers to cope with, adapt to, recover from, or thrive in the face of the challenges of caring. Papers that refer specifically to a process or outcome of resilience (even if proxy measures/concepts are used), and have the enhancement of resilience as an aim, will be included. Papers that focus specifically on interventions to build resilience or on interventions that target a proxy construct for resilience but relate this explicitly to resilience itself will be considered. Papers that simply investigate correlates of resilience (i.e. are not reporting on interventions to enhance resilience) will be excluded. Phenomenon of interest Qualitative studies that aim to describe how interventions to build resilience in family carers are experienced and perceived by family carers, including factors that influence their implementation, will be included. doi: 10.11124/jbisrir-2015-1994 Page 47
Context The review will consider studies conducted in any cultural or geographic context and in any setting including participants homes in the community, residential aged care, or hospital, medical or allied health practice. Outcomes As factors that promote resilience are considered to span biological, personal, social, cultural and environmental domains, the outcomes may be related to any of these domains. Quantitative studies will be included that contain either objective or subjective outcome measures (or a combination of both). Whilst proxy constructs are recognized to be correlated with resilience, and have conceptual overlap with resilience, there is no consensus as to their ability to replace resilience as a construct. Therefore, only those papers using a proxy construct for resilience that explicitly relate the aims of the intervention and the measurement of outcomes to resilience itself will be considered for inclusion. Proxy constructs may include, but are not limited to, self-efficacy, locus of control, perceived burden, psychological wellbeing, strength, coping, positive adjustment and resourcefulness. 11,21,31,32 Studies will be included that contain either objective or subjective outcome measures (or a combination of both) such as questionnaires (e.g. Symptom Distress Questionnaire), surveys (e.g. Personal Views Survey), interviews, self-report inventories (e.g. The Connor-Davidson Resilience Scale 36, The Resilience Scale 37 ), observational assessments, psychological or physiological clinical scales and physiological health measures. Types of Studies The quantitative component will consider for inclusion both experimental and descriptive study designs including randomized controlled trials, non-randomized controlled trials, quasi-experimental, before and after studies, prospective and retrospective cohort studies and cross sectional studies. The qualitative component of the review will consider for inclusion studies that focus on qualitative data including, but not limited to, designs such as phenomenology, grounded theory, ethnography, action research and feminist research. Search strategy The search strategy will aim to find both published and unpublished studies. A three-step search strategy will be used, with an initial limited search of MEDLINE and CINAHL followed by an analysis of title and abstract, and index terms. A second search using all identified keywords and index terms will then be undertaken across databases. Third, the reference list of all articles will be searched for additional studies. Studies published in English in or after 1990 will be considered for inclusion, as an initial search indicated that the majority of intervention studies targeted by this review have been published after this date. The databases to be searched include: CINAHL, PsycInfo, ISI Web of Science, PubMed, Embase, APAIS-Health, Sociological Abstracts, ProQuest Digital Dissertations and ProQuest research library. The search for unpublished studies will include Google Scholar and CareSearch. Initial keywords to be used will be: Population: Alzheimer* OR dement* OR OR Creutzfeldt Jakob Syndrome OR Lewy Body Disease OR Wernicke* OR Korsakoff* OR Huntington* OR Progressive Supranuclear Palsy OR Picks Disease OR Binswanger* doi: 10.11124/jbisrir-2015-1994 Page 48
family OR carer* OR spous* OR daughter OR son OR partner OR informal carer* OR unpaid carer* OR caregiver* OR husband* OR wife OR wives Phenomena of interest: resilien* OR coping OR stress resistance OR stress management OR stress coping OR adaptation OR psychological endurance OR adjustment OR hardiness OR psychological wellbeing OR thriv* OR flourish* OR adaptive behavio$r OR cognitive techniques OR coping behavio$r OR emotional adjustment OR emotional stability OR psychological growth OR self care skills intervention* OR promot** OR program* OR support OR trial OR training OR treat* OR educ* OR manag* Study type: qualitative OR content analysis OR thematic analysis OR field studies OR multi method studies OR mixed method OR experience OR grounded theory OR observ* OR focus group* OR narrative OR action research OR discourse OR phenomenolog* OR ethnograph* OR hermeneutic* OR lived experience OR life experience* OR interpretive synthesis OR interpretive OR interview* OR ethnonursing research OR ethnological research OR feminist critique OR ethosnursing OR ethoslogical research. rct OR "random allocation" OR "randomi?ed control trial" OR "comparative stud*" OR "interrupted time series" OR "clinical trial" OR "prospective stud*" OR "study design" OR "evaluation research" OR "controlled stud*" OR "cohort" OR "case control" OR "interrupted time series" OR "experimental stud*" Assessment of methodological quality Quantitative studies selected for retrieval will be assessed by two independent reviewers for methodological validity prior to inclusion in the review using standardized critical appraisal instruments from the Joanna Briggs Institute Meta-Analysis of Statistics Assessment and Review Instrument (JBI- MAStARI) (Appendix I). Qualitative studies selected for retrieval will be assessed by two independent reviewers for methodological validity prior to inclusion in the review using standardized critical appraisal instruments from the Joanna Briggs Institute Qualitative Assessment and Review Instrument (JBI-QARI) (Appendix I). Any disagreements that arise between the reviewers will be resolved through discussion, or with a third reviewer. Data extraction Quantitative data will be extracted from papers included in the review using the standardized data extraction tool from JBI-MAStARI (Appendix II). The data extracted will include specific details about the interventions, populations, study methods and outcomes of significance to the review question and specific objectives. Qualitative data will be extracted from papers included in the review using the standardized data extraction tool from JBI-QARI (Appendix II). The data extracted will include specific details about the interventions, populations, study methods and outcomes of significance to the review question and specific objectives. doi: 10.11124/jbisrir-2015-1994 Page 49
Data synthesis Quantitative data will, where possible, be pooled in statistical meta-analysis using JBI-MAStARI. All results will be subject to double data entry. Effect sizes expressed as odds ratio (for categorical data) and weighted mean differences (for continuous data) and their 95% confidence intervals will be calculated for analysis. Heterogeneity will be assessed statistically using the standard Chi-square. Where statistical pooling is not possible the findings will be presented in narrative form including tables and figures to aid in data presentation where appropriate. Qualitative research findings will, where possible, be pooled using JBI-QARI. This will involve the aggregation or synthesis of findings to generate a set of statements that represent that aggregation, through assembling the findings rated according to their quality, and categorizing these findings on the basis of similarity in meaning. These categories will then be subjected to a meta-synthesis in order to produce a single comprehensive set of synthesized findings that can be used as a basis for evidence-based practice. Where textual pooling is not possible the findings will be presented in narrative form. Conflicts of interest The authors have no conflicts of interest to declare. Acknowledgements The review is funded by the Dementia Collaborative Research Centers through a DCRC: Carers and Consumers systematic review grant. doi: 10.11124/jbisrir-2015-1994 Page 50
References 1. Alzheimer s Disease International. The global impact of dementia 2013-2050. London: 2013. 2. Access Economics. Making choices, future dementia care: Projects, problems and preferences. Canberra: 2009. 3. Lavretsky H, Siddarth P, Irwin MR. Improving depression and enhancing resilience in family dementia caregivers: A pilot randomized placebo-controlled trial of escitalopram. Am J Geriatr Psychiatry. 2010; 18, 2: 154-62. 4. Clark MC, Diamond PM. Depression in family caregivers of elders: A theoretical model of caregiver burden, sociotropy, and autonomy. Res Nurs Health. 2010; 33, 1: 20-34. 5. Ornstein K, Gaugler JE. The problem with problem behaviors : A systematic review of the association between individual patient behavioral and psychological symptoms and caregiver depression and burden within the dementia patient caregiver dyad. Int Psychogeriatr. 2012; 24, 10: 1536-52. 6. O Dwyer ST, Moyle W, Pachana NA, Sung B, Barrett S. Feeling that life is not worth living (death thoughts) among middle-aged, Australian women providing unpaid care. Maturitas. 2014; 77, 4: 375-9. 7. O'Dwyer S, Moyle W, van Wyk S. Suicidal ideation and resilience in family carers of people with dementia: A pilot qualitative study. Aging Ment Health. 2013 2013/08/01; 17, 6: 753-60. 8. Sörensen S, Duberstein P, Gill D, Pinquart M. Dementia care: Mental health effects, intervention strategies, and clinical implications. Lancet Neurol. 2006; 5, 11: 961-73. 9. Scott CB. Alzheimer's disease caregiver burden: Does resilience matter? J Hum Behav Soc Environ. 2013 2013/11/01; 23, 8: 879-92. 10. Slattum P, Johnson MA. Caregiver burden in alzheimer's disease. Consult Pharm. 2004; April 2004, 4: 352-62. 11. Contador I, Fernández-Calvo B, Palenzuela DL, Miguéis S, Ramos F. Prediction of burden in family caregivers of patients with dementia: A perspective of optimism based on generalized expectancies of control. Aging Ment Health. 2012; 16, 6: 675-82. 12. Thommessen B, Aarsland D, Braekhus A, Oksengaard AR, Engedal K, Laake K. The psychosocial burden on spouses of the elderly with stroke, dementia and Parkinson's disease. Int J Geriatr Psychiatry. 2002; 17, 1: 78-84. 13. O'Dwyer ST, Moyle W, Zimmer-Gembeck M, De Leo D. Suicidal ideation in family carers of people with dementia: a pilot study. Int J Geriatr Psychiatry. 2013; 28, 11: 1182-8. 14. Schulz R, Beach S. Caregiving as a risk factor for mortality: The caregiver health effects study. J Am Med Assoc. 1999; 282, 23: 2215. 15. Cherry M, Salmon P, Dickson J, Powell D, Sikdar S, Ablett J. Factors influencing the resilience of carers of individuals with dementia. Rev Clin Gerontol. 2013; 23, 04: 251-66. 16. Herrman HMD, Stewart DEMD, Diaz-Granados NM, Berger ELD, Jackson BP, Yuen TB. What is resilience? Can J Psychiatry. 2011 May 2011; 56, 5: 258-65. 17. Zautra AJ. Resilience: One part recovery, two parts sustainability. J Pers. 2009; 77, 6: 1935-43. doi: 10.11124/jbisrir-2015-1994 Page 51
18. Ong AD, Bergeman CS, Boker SM. Resilience comes of age: Defining features in later adulthood. J Pers. 2009; 77, 6: 1777-804. 19. Zauszniewski JA, Bekhet AK, Suresky MJ. Resilience in family members of persons with serious mental illness. Nurs. Clin. North Am. 2010; 45, 4: 613-26. 20. O Rourke N, Kupferschmidt AL, Claxton A, Smith JZ, Chappell N, Beattie BL. Psychological resilience predicts depressive symptoms among spouses of persons with Alzheimer disease over time. Aging Ment Health. 2010 2010/11/01; 14, 8: 984-93. 21. Gaugler JE, Kane RL, Newcomer R. Resilience and transitions from dementia caregiving. J Gerontol B Psychol Sci Soc Sci. 2007 January 1, 2007; 62, 1: P38-P44. 22. Jang H, Yi M. Effects of burden and family resilience on the family adaptation of family caregivers of elderly with dementia. Korean Journal of Adult Nursing. 2013; 25, 6: 725-35. 23. Heisel MJ, Flett GL. Psychological resilience to suicide ideation among older adults. Clin Gerontol. 2008 2008/05/30; 31, 4: 51-70. 24. Johnson J, Wood AM, Gooding P, Taylor PJ, Tarrier N. Resilience to suicidality: The buffering hypothesis. Clin Psychol Rev. 2011; 31, 4: 563-91. 25. Alzheimer's Australia. First steps to a dementia friendly Australia. Canberra: 2014. 26. Australian Government Department of Social Services. Aged care reform: Australian Government; No date [cited 2014 19 December]. Available from: https://www.dss.gov.au/ourresponsibilities/ageing-and-aged-care/aged-care-reform. 27. Kidd LI. The effect of a poetry writing intervention on self-transcendence, resilience, depressive symptoms, and subjective burden in family caregivers of older adults with dementia: Case Western Reserve University; 2009. 28. Harris PB. Another wrinkle in the debate about successful aging: The undervalued concept of resilience and the lived experience of dementia. Int J Aging Hum Dev. 2008; 67, 1: 43-61. 29. Wilks SE, Croom B. Perceived stress and resilience in Alzheimer's disease caregivers: Testing moderation and mediation models of social support. Aging Ment Health. 2008 2008/05/01; 12, 3: 357-65. 30. Fitzpatrick KE, Vacha-Haase T. Marital satisfaction and resilience in caregivers of spouses with dementia. Clin Gerontol. 2010 2010/05/28; 33, 3: 165-80. 31. Bekhet AK. Effects of positive cognitions and resourcefulness on caregiver burden among caregivers of persons with dementia. Int J Ment Health Nurs. 2013; 22, 4: 340-6. 32. Dias Lopes da Rosa R, Santos RL, Barroso de Sousa MF, Moreira Lima Nogueira M, Torres B, Belfort T, et al. Resilience of caregivers of people with dementia: a systematic review of biological and psychosocial determinants. Trends Psychiatry Psychother. 2015; 31, 1: 12-19. 33. Pinquart M, Sörensen S. Helping caregivers of persons with dementia: Which interventions work and how large are their effects? Int Psychogeriatr. 2006; 18, 04: 577-95. 34. Sörensen S, Pinquart M, Duberstein P. How effective are interventions with caregivers? An updated meta-analysis. Gerontologist. 2002 June 1, 2002; 42, 3: 356-72. doi: 10.11124/jbisrir-2015-1994 Page 52
35. Canadian Palliative Care Association. Standards for palliative care provision. Ottawa: Canadian Palliative Care Association: 1998. 36. Connor KM, Davidson JR. Development of a new resilience scale: the Connor-Davidson Resilience Scale (CD-RISC). Depress Anxiety. 2003; 18, 2: 76-82. 37. Wagnild GM, Young HM. Development and psychometric evaluation of the reslience scale. J Nurs Meas. 1993; 1, 2. doi: 10.11124/jbisrir-2015-1994 Page 53
Appendix I: Appraisal instruments QARI appraisal instrument Insert page doi: 10.11124/jbisrir-2015-1994 Page 54
MAStARI appraisal instrument Insert doi: 10.11124/jbisrir-2015-1994 Page 55
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Appendix II: Data extraction instruments QARI data extraction instrument doi: 10.11124/jbisrir-2015-1994 Page 58
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MAStARI data extraction instrument doi: 10.11124/jbisrir-2015-1994 Page 60
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